United States

Background:
Acquired hemophilia A (AHA) is an autoimmune disease characterized by the development of autoantibodies against coagulation factor VIII (FVIII). AHA most commonly occurs spontaneously in older adults, but can be triggered by malignancy, autoimmune disease, pregnancy, infection, and drugs, particularly antibiotics. We report a rare case of AHA diagnosed in the setting of GI bleeding refractory to numerous endoscopic treatments.

Case Presentation:
A 71-year-old man with coronary artery disease who was initially hospitalized for cellulitis that did not respond to multiple prolonged courses of outpatient antibiotics developed a GI bleed. On admission, he was placed on cefazolin and developed hematemesis 4 days later. Endoscopy identified an oozing gastroesophageal junction (GEJ) ulceration with a visible blood vessel. It was treated with epinephrine injections, hemostatic clips, and spray. The patient was readmitted twice shortly after discharge for recurrent bleeding, first for melena and then for hemorrhagic shock. He received four total endoscopies finding active bleeding at the GEJ, all with successful procedural hemostasis. Coagulopathy workup revealed a persistently elevated prothrombin time, with only partial correction in mixing studies. Further testing identified the specific FVIII inhibitor, quantified its high titer of 71 Besthesda Units, and confirmed extremely low FVIII activity (&lt;1%) diagnostic for an acquired FVIII deficiency. Sulfasalazine was considered the most likely inducer. The bleeding stopped after fresh frozen plasma transfusions, and he was discharged home on corticosteroids, with plans to start rituximab.

Discussion:
This case underscores the consideration of coagulopathies as possible contributors to refractory anemia and GI bleeding. Recent antibiotics should increase suspicion of AHA, even without a prior history of abnormal coagulation. This approach can lead to timely and appropriate treatment interventions, such as immunosuppressive therapy and FVIII replacement, ultimately improving patient outcomes. Despite its rarity, awareness and suspicion of AHA can significantly impact the management and prognosis of affected patients.


AMA Research Challenge 2024 

Acquired Hemophilia A: A Rare Case of Persistent Peptic Ulcer Bleeding Despite Multiple Endoscopic Treatments


Background:
Acquired hemophilia A (AHA) is an autoimmune disease characterized by the development of autoantibodies against coagulation factor VIII (FVIII). AHA most commonly occurs spontaneously in older adults, but can be triggered by malignancy, autoimmune disease, pregnancy, infection, and drugs, particularly antibiotics. We report a rare case of AHA diagnosed in the setting of GI bleeding refractory to numerous endoscopic treatments.

Case Presentation:
A 71-year-old man with coronary artery disease who was initially hospitalized for cellulitis that did not respond to multiple prolonged courses of outpatient antibiotics developed a GI bleed. On admission, he was placed on cefazolin and developed hematemesis 4 days later. Endoscopy identified an oozing gastroesophageal junction (GEJ) ulceration with a visible blood vessel. It was treated with epinephrine injections, hemostatic clips, and spray. The patient was readmitted twice shortly after discharge for recurrent bleeding, first for melena and then for hemorrhagic shock. He received four total endoscopies finding active bleeding at the GEJ, all with successful procedural hemostasis. Coagulopathy workup revealed a persistently elevated prothrombin time, with only partial correction in mixing studies. Further testing identified the specific FVIII inhibitor, quantified its high titer of 71 Besthesda Units, and confirmed extremely low FVIII activity (<1%) diagnostic for an acquired FVIII deficiency. Sulfasalazine was considered the most likely inducer. The bleeding stopped after fresh frozen plasma transfusions, and he was discharged home on corticosteroids, with plans to start rituximab.

Discussion:
This case underscores the consideration of coagulopathies as possible contributors to refractory anemia and GI bleeding. Recent antibiotics should increase suspicion of AHA, even without a prior history of abnormal coagulation. This approach can lead to timely and appropriate treatment interventions, such as immunosuppressive therapy and FVIII replacement, ultimately improving patient outcomes. Despite its rarity, awareness and suspicion of AHA can significantly impact the management and prognosis of affected patients.

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Background
Small intestinal bacterial overgrowth (SIBO) is a condition that is defined by an overgrowth of normal flora in the small intestinal, causing symptoms similar to irritable bowel disease, such as abdominal pain, diarrhea, and frequent stools. Altered flora can arise from dysmotility, altered anatomy, such as surgery, or hypochlorhydria. A known condition that is associated with SIBO is auto-brewery syndrome, in that the intestinal flora metabolize carbohydrates into ethanol, which will, then, intoxicate the patient. However, alcohol abuse is a common condition worldwide. Patients that are addicted to alcohol often have financial, social, and interpersonal strain, and those patients often go to great lengths to hide their alcohol abuse to their spouses and family, often involving elaborate lies and trickery. Moreover, a patient that has a history of alcohol abuse is at greater risk of relapse following abstinence from alcohol.
Case Presentation
We present a case of a 48-year-old patient with a medical history of SIBO who is presenting to the emergency department acutely intoxicated. The patient is accompanied by his wife. She states that since he has been diagnosed with SIBO, he often experiences multiple “episodes” per month where he appears intoxicated, and that it is controlled by a specific diet that they have worked to develop. She endorses that he has a history of alcohol abuse, and that he has abstained from alcohol for about 2 years, with no possibility of the patient relapsing as they both work from home and the majority of their time together. Labs were significant for a blood alcohol level (BAC) of 0.47. He was then admitted to the hospital, and his BAC was trended, which showed a linear decrease of .02 per hour. The patient was not restricted in his diet and was seen to eat carbohydrates, with no increase in BAC, thus ruling out auto-brewery syndrome, leaving a relapse of alcohol abuse as his final diagnosis.
Discussion
This case provides an intricate display of SIBO, auto-brewery, and chronic alcohol abuse disorder. The endogenous production of ethanol by intestinal microorganisms results in intoxication symptoms. The differentiation of auto-brewery and alcohol use is crucial as management includes carbohydrate-restricted diet, antibiotics, and cessation of alcohol consumption. Psychosocial elements of this case highlight the interdepartmental teamwork from medical doctors, nutritionists, and psychosocial personnel and support systems.

Alcohol Abuse or Auto-Brewery A Case of Complicated SIBO and Addiction

Background
Emphysematous gastritis is a rare and lethal disease characterized by air within the gastric wall, caused by gas forming microorganisms such as Escherichia coli, streptococcus species, and clostridium species, to name a few. Even when identified early, it has a mortality rate of 60%. Here we present a rare case of emphysematous gastritis that developed while the patient was admitted to the hospital for an unrelated reason.

Case Presentation
A 62-year-old male with past medical history of type 2 diabetes mellitus and osteoarthritis with long-term naproxen use presented to the hospital for an elective left total knee arthroplasty, which he underwent successfully. On postoperative day 3, patient developed dyspnea and severe abdominal pain accompanied by nausea and multiple episodes of nonbilious emesis. On exam, abdomen was moderately distended with guarding and diffuse tenderness to palpation, but bowel sounds were preserved. Lab studies remarkable for white blood cell count 13.7x103/L, hemoglobin 9.7 g/dL, and lactic acid 1.5 mEq/L. An emergent CT scan with IV contrast showed evidence of emphysematous gastritis with portal venous gas. Due to the high risk of iatrogenic perforation from endoscopy, he was managed non-operatively with IV fluids, pantoprazole, antiemetics, and antibiotic coverage with vancomycin and piperacillin-tazobactam. Within 48 hours, the patient’s symptoms resolved, blood cultures were negative, and he was discharged shortly afterwards to finish a 14-day course of levofloxacin and metronidazole.

Discussion
It is often challenging to distinguish between emphysematous gastritis and gastric emphysema. Gastric emphysema is an asymptomatic non-infectious process that is associated with mechanical disruption of the gastric wall (i.e., surgery, endoscopy or nasogastric tube placement). In contrast, emphysematous gastritis is a life-threatening condition often caused by a bacterial infection that arises locally or spreads hematogenously from a distant focus. In our case, the major risk factors were diabetes mellitus and chronic nonsteroidal anti-inflammatory drug use, creating a favorable environment for bacterial invasion. Although the mainstay of treatment is supportive, emphysematous gastritis can lead to severe septic shock, gastric perforation, hemorrhage, or gastric wall necrosis if not identified promptly. In such cases, emergent surgical intervention is warranted.


Emphysematous Gastritis: A Rare but Critical Gastrointestinal Condition


Background
Hydatid cyst disease is amongst the most common zoonotic parasitic manifestations, caused by Echinococcus granulosus where humans act as an accidental host. Hydatid cyst of anterior wall (1-4%) has been observed in a few patients without primary involvement of the liver. 

Case Presentation
A 33 year old, young male presented with on and off dull aching abdominal pain with fever for 5 years but was misdiagnosed as anterior abdominal wall haematoma. The only significant history was possession of an unvaccinated stray dog. Physical examination revealed a cystic, tender swelling around 5x10 cm in the region of right hypochondrium to right lumbar region. Abdominal Contrast enhanced computed tomography revealed a slightly thick walled cystic appearing lesion 6.1x 9.6 x 5.5 cm suggesting a focal discontinuity of its antero-medial wall with surrounding relatively ill- defined hypo-dense collection seen partly insulating in the inter-muscular planes, suggesting a partly ruptured hydatid cyst without any evidence of any intraperitoneal extension. Serology of Hydatid cyst was positive. Patient was prophylactically started on Albendazole until surgical exploration. During surgery, cystic mass was found adherent to anterior abdominal wall muscles without any parietal peritoneum adhesions. En-masse excision was done from subcutaneous planes, keeping the cyst wall intact. The point of rupture was identified and the cyst fluid and daughter cysts suctioned with minimal spill to vicinity and irrigation was performed with hypertonic saline. Cetrimide soaked gauze was placed at operative site for 10 minutes. Gross and Histopathology examination confirmed Hydatid cyst. Patient was advised to take oral Albendazole and Praziquantel for 6 months. Patient had an uneventful post-operative course till 6 months follow up.

Discussion 
Echinococcus granulosus, the smallest of all tapeworms, a zoonotic cestode with humans being an accidental host infected by ingestion or direct contact with infected canines. The clinical course and symptoms of cystic hydatidosis depend upon site of infection, size of cysts and the pressure symptoms caused by enlargement of the cysts. Careful history taking, thorough examination along with the use of radiological and serological investigations aid in reaching a confirmatory diagnosis of the disease. Treatment modalities comprise of anti- parasitic drugs, PAIR (puncture-aspirate-inject- reaspirate) and surgery. Prevention of cystic echinococcosis includes public education, deworming of dogs and slaughter house hygiene. Early and prompt diagnosis with timely management is the need of the hour to decrease the global burden of the disease. 


Hydatid Cyst of Anterior Abdominal Wall: A Case Report

A 56-year-old woman with a past medical history of alcohol-associated cirrhosis presented for hematochezia. She was hypotensive. A computed tomography (CT) angiogram scan of the abdomen and pelvis revealed a conglomeration of severe right lower quadrant (RLQ) varices extending to the right colon wall. The prevalence of colonic varices (CV) is likely underestimated. While gastrointestinal bleeding from colonic varices is rare, there is a potential risk of death in severe cases. Currently, there are no specific guidelines for managing colonic varices. The management choice is based on the etiology, severity of bleeding, and distribution of the varices.

Massive Lower Gastrointestinal Bleeding From Colonic Varices In a Patient With Cirrhosis

Introduction: Diverticulitis is a well-studied colorectal disease that is mostly managed conservatively. Severe diverticulitis may progress to pericolic abscess or perforation when not caught early. Patients with diverticulitis are predisposed to hepatic abscesses via seeding through the portal system. In rare cases such as ours, the hepatic abscesses lead to a hepatopulmonary fistula, causing recurrent symptomatic pleural effusions. 

Case Description: A 61-year-old male with history of right sided diverticulitis presented to the emergency department with a 2-week history of worsening abdominal pain and a 2-day history of chest pain and dyspnea. On physical exam, he was diaphoretic, tachycardic, and hypotensive. Chest X-Ray revealed a moderate sized right sided pleural effusion. CT scan of Abdomen Pelvis with IV Contrast was indicative of multiple large air fluid collections surrounding the liver with one collection near the dome of the liver that fistulized to the right lower lobe of the lung. There was also notable wall thickening of the cecum and ascending colon with surrounding inflammation, suggesting diverticulitis. Due to his hemodynamic status, sepsis protocol was initiated, including treatment with broad spectrum IV antibiotics. CT guided drainage catheter was placed within the liver abscess and intracatheter contrast confirmed a fistulous tract into the right lower lobe of the lung. A chest tube was placed to drain the pleural effusion, and fluid culture was positive for S. Anginosus, B. Fragillis, and Prevotella, indicating a clear abdominal source. The patient’s hospital course was prolonged due to the refractory nature of his pleural effusions, but with targeted antibiotic therapy and respiratory support, he improved and was discharged with outpatient IV antibiotic therapy for total of 6 weeks. 

Discussion: Up to 25% of patients with diverticulitis develop complications. While some complications such as microperforations cause contained pericolic abscesses, macroperforations cause colo-colonic fistulas or widespread abscesses throughout the abdomen, including the liver. If left untreated, it can lead to fulminant sepsis, significantly worsening the patient’s prognosis. In our case, the CT scan was crucial in identifying the hepatopulmonary fistula early, allowing us to drain the abscess promptly. Surgical decortication would have likely been pursued if patient had not improved with time. 


Recurrent Pleural Effusions from Hepatopulmonary Fistula Secondary to Perforated Diverticulitis

Abstract:
Background: Malaria caused by Plasmodium vivax is often considered less severe than infections caused by other Plasmodium species. However, recent evidence suggests that P. vivax can lead to severe complications, including hepatic dysfunction and transudative ascites. This case report describes an unusual presentation of severe P. vivax malaria in a 14-year-old female, characterized by persistent vomiting, loose stools, jaundice, and high-grade fever with rigors.
Case Presentation: The patient, who had a history of acute gastroenteritis, was admitted with symptoms persisting for 8 days. Initial findings included anemia, elevated liver enzymes, and transudative ascites. Diagnostic imaging revealed splenomegaly, an enlarged liver, and mild ascites. Ascitic fluid analysis was consistent with transudative ascites and showed elevated adenosine deaminase (ADA) levels. The diagnosis of P. vivax malaria was confirmed by identifying gametocytes in the peripheral blood smear. Treatment with intravenous Sulbactam and Artemisinin resulted in significant clinical improvement.
Discussion: This case highlights the potential for severe manifestations of P. vivax malaria, including jaundice, hepatic dysfunction, and transudative ascites. The patient's presentation underscores the need for a high index of suspicion for malaria in endemic regions, especially when clinical symptoms overlap with other tropical diseases. The resolution of symptoms with appropriate treatment supports the diagnosis and emphasizes the importance of timely intervention.

Unraveling the connection between Vivax Malaria and Transudative Ascites. A case report and literature review

Abstract Title: Unveiling the Cholestatic Phenotype: A Case of Autoimmune Hepatitis with Severe Hyperbilirubinemia 

Background: Autoimmune hepatitis (AIH) is defined by clinical, laboratory, and histologic findings as a hepatitic syndrome rather than a cholestatic syndrome. Cholestasis in AIH is typically due to associated primary biliary cirrhosis or primary sclerosing cholangitis. However, 8% cases of AIH have unexplained cholestasis (the Cholestatic-phenotype). In this case report, we described a patient who presented with cholestasis but was later diagnosed with AIH, responding well to steroid treatment. 

Case Presentation: A 52-year-old woman presented to the emergency department with a 15-day history of yellowing of the eyes and urine, accompanied by fatigue and pale-stools, following a brief febrile illness. Initial treatment with empirical antibiotics and supportive care at a local health center showed no improvement. Her medical history was unremarkable except for a similar episode of jaundice 4 years prior. Physical examination was normal, but laboratory tests revealed normocytic normochromic anemia, markedly elevated bilirubin (total 27 mg/dl, direct 15 mg/dl, indirect 12 mg/dl), and liver enzymes (AST-239 IU/L, ALT - 93 IU/L). Further evaluation showed a positive antinuclear antibody (ANA) profile with antibodies against SS-A, Scl-70 and Jo-1, but negative for anti-LKM, anti SMA and AMA. Liver ultrasound revealed altered liver echotexture without strictures or other abnormalities. Liver biopsy demonstrated features of auto-immune hepatitis(Figure 1), with interface hepatitis, cholestasis, chronic inflammatory infiltrates and portal fibrosis. Based on the Revised original scoring system for AIH, the patient had a pretreatment score of 15 (Simplifed scoring system score of 8) ,confirming the diagnosis. Treatment with azathioprine and prednisolone was initiated, with subsequent follow-up showing a positive response. Bilirubin levels normalized within two months. 

Discussion: For patients with cholestasis, especially with elevated bilirubin and positive ANA profile, AIH should be one of the differential diagnosis. Even if the classic serological markers like anti LKM and anti-SMA are negative, biopsy is the key to confirm the diagnosis in patients with such atypical presentation. The standard treatment for AIH is immunosuppressive therapy using steroids and steroid sparing agent such as azathioprine. Steroids such as prednisolone reduces the inflammation acutely and azathioprine is used for long-term chronic therapy to maintain the remission without the fear of steroid dependence.

Unveiling the Cholestatic Phenotype: A Case of Autoimmune Hepatitis with Severe Hyperbilirubinemia

Background:
Peripheral artery disease is the narrowing or blockage of arteries in the peripheral circulation, primarily caused by atherosclerosis. Treatment may initially be an exercise program and a focus on reducing risk factors such as quitting smoking and treating comorbid conditions. The next step is revascularization procedures, often starting with an endovascular approach (balloon angioplasty with or without stenting) and proceeding to surgical interventions if endovascular approach fails or is contraindicated. Surgical procedures include anatomical bypasses, such as aortofemoral bypass grafting, and extra-anatomical bypasses, such as axillofemoral bypass grafting (AXFBG).

Case Presentation:
A 67-year-old female with HTN, HLD, CVA with residual L sided deficits, PAD s/p L fem-AT bypass with rGSV (2020) with subsequent L CFA endarterectomy (2020), L groin debridement (2021) and recent LLE angiogram at OSH (2/5/24) who presented with acute on chronic LLE critical limb ischemia and found to have necrotic left great toe. CTA with occlusion of L common and external iliac artery with possible dissection in L CFA with reconstitution in L SFA. Preoperative diagnosis was left lower extremity ischemic ulceration. The patient underwent left axillary artery to proximal superficial femoral artery (axillofemoral) bypass 2/27/24. Patient was
recovering well after surgery until post op day 3. Patient developed symptoms of post ischemic compartment syndrome. Patient had pain out of proportion on left leg, extreme tenderness to touch, worse pain with passive extension, and some swelling in the left lower extremity. Palpable pulses were unchanged from before the surgery. Left dorsalis pedis pulse was not palpable and not detected with doppler at baseline. Ultimately, vascular surgery recommended left above knee amputation, which was done on 3/11/24.

Discussion:
Post-ischemic compartment syndrome is a surgical emergency caused by increased
intercompartmental pressure in the lower extremity that develops following revascularization procedures for acute limb ischemia. Surgical revascularization can cause the muscles of the extremity to develop edema due to fluid extravasation or inflammatory responses following an ischemia-reperfusion injury, resulting in rapid increase in ICP. Common procedures that cause this are embolectomy or arterial bypass. Inadequate backflow, high serum CK level, positive fluid balance, and advanced-stage acute limb ischemia are predictive factors for developing compartment syndrome. This patient had an amputation instead of a fasciotomy because the limb was already not being perfused well. If she had a fasciotomy, it would not have healed well and could have put the patient at risk for infection.

Axillofemoral Bypass and Post-Reperfusion Compartment Syndrome: Case Report

Biodegradable Temporizing Matrices (BTMs) are temporary, biocompatible, and resorbable scaffolds designed to temporarily occupy a defect site, facilitating tissue regeneration and neodermal growth in a surgical wound or defect site while minimizing the need for extensive tissue transfers or permanent implants. Spiradenomas are mostly benign skin tumors developing from sweat gland cells that typically require surgical excision as treatment, which can result in scarring and functional impairment, especially in functional joint areas like the ankle. While the application of BTMs in small- to moderate-sized defects resulting from malignant resections in functionally critical areas has shown promising results in preliminary studies, the use of BTM within the context of resection margins is an emerging concept that needs to be further studied. 
This case report presents the successful application of a BTM in the reconstruction of a soft tissue defect following wide local excision of a spiradenoma on the right ankle of a 64-year-old male patient, demonstrating the value of these matrices as an essential reconstructive tool in the management of soft tissue defects following malignant resections.

Biodegradable Temporizing Matrix (BTM) in Small Scale Reconstruction of Malignant Resections in Functional Areas: A Case Report

Introduction
Surgical drains are crucial in postoperative management to prevent complications such as fluid collections and abscess formation. However, they carry risks like migration or erosion into adjacent organs, leading to serious consequences like gastric outlet obstruction. This case highlights the complexities of managing a surgical drain eroding into the duodenum following interventions for gallstone pancreatitis.

Case Description
A 76-year-old male with a history of benign prostatic hyperplasia, hypertension, gastroesophageal reflux disease, and recent gallstone pancreatitis presented to the ED with persistent vomiting and intolerance to oral intake. Initial assessment showed tachycardia, hypotension, and hypoxemia. He had undergone a laparoscopic cholecystectomy converted to open subtotal cholecystectomy and pancreatic necrosectomy 50 days earlier, with a Jackson-Pratt (JP) drain placed for purulent fluid management. He presented with worsening symptoms, including abdominal pain, purulent JP drain output, and a 30-pound weight loss. Severe protein-calorie malnutrition was noted, and total parenteral nutrition (TPN) was started. Lab findings indicated anemia of chronic disease.
CT imaging revealed gastric dilation, duodenal narrowing, and wall thickening suggesting gastric outlet obstruction from postoperative sequelae or pancreatitis inflammation. Additional findings included esophagitis, walled-off necrosis, and infected collections around the pancreas. Endoscopies on days 3 and 10 identified severe esophagitis and JP drain migration into the duodenum, causing obstruction. The drain was repositioned, resolving the obstruction and allowing diet progression.
By day 14, an upper GI study confirmed no duodenal leaks, enabling a transition to a regular diet and discharge on day 16. 

Discussion
This case underscores the complexities of managing surgical drain complications following extensive interventions for severe gallstone pancreatitis. The patient's history and surgical interventions predisposed him to drain-related complications due to extensive tissue manipulation and inflammatory responses. Endoscopic interventions were crucial for repositioning the drain and resolving the obstruction, highlighting advanced endoscopic techniques' evolving role.
Despite advancements, drain migration remains a concern, emphasizing the need for vigilant postoperative monitoring and early intervention. The patient's malnutrition and anemia added complexity to his recovery, with TPN playing a crucial role in optimizing outcomes.
Recent studies advocate for minimally invasive techniques for necrotizing pancreatitis, showing improved outcomes and reduced complications. Early intervention in pancreatic fluid collections may improve outcomes without increasing morbidity, challenging previous delayed intervention paradigms.
This case emphasizes conservative management and interdisciplinary teamwork in achieving favorable outcomes. Standardized protocols for prevention, early detection, and optimal management are crucial, along with continued research to refine techniques and establish evidence-based guidelines for high-risk scenarios.

Next from AMA Research Challenge 2024

Alcohol Abuse or Auto-Brewery A Case of Complicated SIBO and Addiction

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